Feasibility of a Serious Illness Communication Program for Pediatric Advance Care Planning

Key Points Question Is a structured pediatric advance care planning (ACP) intervention feasible and acceptable and is it associated with improved family-centered outcomes? Findings In this cohort study including 40 interprofessional clinicians, 36 parents, and 6 adolescents and young adults with serious illness, the structured ACP intervention was both feasible and acceptable. Parents reported higher therapeutic alliance immediately after the intervention and decreased anxiety, which persisted at the 1-month follow up. Meaning These findings suggest that this structured advanced care planning intervention was feasible, acceptable, and helpful to clinicians and parents of pediatric patients who are seriously ill.


Introduction
Children and adolescents and young adults (AYAs) with serious illness often have a variable clinical course with periods of stability alternating with life-threatening deteriorations; consequently, many children and AYAs experience health crises without opportunities to discuss preferences for medical care.Furthermore, bereaved parents report a lack of preparation to address their child's medical and emotional needs at end of life (EOL). 1,22][13][14][15] In a 2019 study from our group, ACP was also associated with superior parent-reported EOL care outcomes, including improved preparedness for EOL, the ability to plan their child's location of death, and superior quality of life for their child. 16Additionally, ACP that included specific assessment of family goals was associated with decreases in perceived child suffering at EOL and parental decisional regret. 16[24][25] Multifaceted interventions combining educational and systems-based approaches can increase timely ACP discussions and improve patient and clinician experiences. 10,26,279][30] However, there is limited research on use of these guides in pediatrics to support clinician-led ACP.To address this gap, the Pediatric Serious Illness Communication Program (PediSICP) was designed using a stakeholder-driven formative research approach. 24The PediSICP is a structured ACP intervention consisting of clinician training preceding an ACP communication occasion supported by evidence-based communication guides and a template for electronic medical record (EMR) documentation. 24This pilot study aims to evaluate the feasibility and acceptability of the PediSICP with exploration of outcomes associated with program participation, including therapeutic alliance, shared decision-making, communication quality, depression, and anxiety.

Study Design and Population
The protocol for this cohort study was approved by the Boston Children's Hospital institutional review board.All clinicians and parents provided verbal informed consent to participate, and AYAs provided verbal assent or consent.This study was designed as a mixed-methods, pragmatic, singlegroup, single-institution feasibility cohort pilot and was conducted at a quaternary care children's hospital from April 2021 to March 2023.Here, we report on quantitative study results.Reporting followed the Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) reporting guideline for cohort studies.Children and AYAs with serious illness were identified using Feudtner and colleagues definition of complex chronic conditions. 31AYAs aged 13 years and older with serious illness and parents of children with serious illness who could read and speak English and had at least 2 readmissions in 1 year or 1 admission lasting more than 2 weeks were eligible to participate.Interprofessional clinicians were eligible if they practiced in an inpatient location or specialty that cares for children and AYAs who are seriously ill.

Enrollment and Study Procedures
Eligible clinicians were identified through a hospital database, invited once by email to participate, and enrolled from April to December 2021.Parents and AYAs were approached for enrollment between July 2021 and December 2022.A trained research assistant screened for eligible inpatient AYA and parents daily in the intensive care inpatient units and outpatient clinics of enrolled clinicians.
For outpatients, participants were also identified through referral from enrolled clinicians.On recruitment, parents and AYAs were provided study information and made aware that an ACP conversation was the primary study intervention.To better understand the unique perceptions of the PediSICP, we did not enroll AYA-parent dyads and enrolled only 1 parent per family; however, both parents could participate in ACP conversations.Trained clinicians were notified when AYAs or parents they were caring for were enrolled.ACP conversations were conducted in-person either during admission with inpatient clinicians at a time convenient for the AYA or parent or at a previously scheduled clinic appointment for outpatient clinicians.Following the conversation, clinicians documented discussion content in the EMR using the PediSICP ACP note template. 24All participants received a 1-time retail gift card following intervention completion.

Clinician Training
In keeping with COVID-19 restrictions, 10 trainings, each with 4 to 8 interprofessional participants, were conducted virtually using Zoom video conferencing software (Zoom Video Communications) from April to December 2021.The 3-hour PediSICP clinician training consisted of a brief didactic session on the value of ACP, demonstration and discussion of the PediSICP, and associated EMR documentation, followed by skills practice using a trained actor along with individual coaching from facilitators with serious illness communication training. 32

Data Collection and Survey Instruments
Clinicians completed surveys via electronic data capture before training and after the intervention following their first documented ACP conversation with an enrolled AYA or parent to measure intervention feasibility and acceptability and to explore changes in moral distress using the Moral Distress Scale-Revised (MDS-R). 33Preintervention and postintervention AYA and parent surveys were used to measure the intervention's acceptability using an adaptation of the Lyon Satisfaction Questionnaire, which was developed to assess reactions to an ACP program. 18Surveys were also used to evaluate parent exploratory outcomes at 3 time points: before PediSICP intervention (T1), 2 to 7 days after the intervention (T2), and at 1-month follow-up (T3), including parent report of shared decision-making (assessed using CollaboRATE-5, 34 consisting of 3 items with 5-point Likert responses assessing patients' perceptions of their involvement in decision-making, with higher scores indicating higher shared decision-making), therapeutic alliance (assessed using The Human Connection [THC] scale, 35 consisting of 6 items with 4-point Likert responses assessing patientperceived therapeutic alliance, with scores ranging from 0-64 and higher scores reflecting stronger therapeutic alliance), communication quality (assessed using the Quality of Communication Questionnaire [QOC], 36 a 17-item survey measuring general and EOL care communication skills on a scale from 0-10 with higher scores indicating higher communication quality), depression (assessed using the Patient Health Questionnaire 9-item scale [PHQ-9], 28 a 27-point scale where scores of 5, 10, 15, and 20 represent mild, moderate, moderately severe, and severe depression, respectively), and anxiety (measured using the Generalized Anxiety Disorder 7-item scale [GAD-7], 37 with scores ranging from 0-21 and higher scores indicating higher levels of anxiety, with cut points of 5 considered mild anxiety; 10, moderate anxiety; and 15, severe anxiety).

Outcome Measures
Our primary aims were to assess feasibility and acceptability.Feasibility was defined a priori as at least 70% of enrolled clinicians completing training and having at least 1 documented ACP conversation with an enrolled AYA or parent using the PediSICP framework.This threshold is  [38][39][40] We also assessed acceptability of the PediSICP among AYAs, parents, and interprofessional clinicians.Our primary acceptability end point was defined as at least 70% of parent participants would agree or strongly agree that the experience was worthwhile, that they felt listened to, and that they would recommend the intervention to other families, as measured on postintervention surveys.For clinicians, acceptability was defined as at least 70% of postintervention survey respondents would agree or strongly agree that they felt prepared for the ACP conversation, the guide was useful, and they would recommend the PediSICP to colleagues.This pilot study was not designed or powered to test intervention efficacy; however, to collect data for future clinical trials, exploratory outcomes were evaluated at 3 time points, as previously described.

Statistical Analysis
Self-reported participant characteristics, including race and ethnicity, were collected at enrollment, and reactions to the PediSICP were collected on the post-PediSICP survey and summarized using descriptive statistics.

Clinician Participation in the PediSICP Training and Feasibility
We contacted 137 clinicians for participation in the PediSICP training; 40 clinicians enrolled in the study by completing the pretraining survey and participating in 1 of 10 virtual trainings (Figure 1).
Thirty enrolled clinicians (75%), including 20 physicians, 6 nurse practitioners, 3 registered nurses, 1 respiratory therapist, completed the PediSICP intervention by having and documenting an ACP conversation with an enrolled parent or AYA, thus meeting our feasibility goal.There were no significant differences in clinician characteristics between enrolled clinicians that completed the intervention and those who did not.Reasons for noncompletion were primarily due to the lack of an enrolled AYA or parent during a clinician's service week or clinic time.

Parent Participation in PediSICP Intervention
We screened 96 parents, of whom 73 were eligible and 46 enrolled, and 36 enrolled parents participated in an ACP conversation using the PediSICP framework, 33 parents completed the T2 survey, and 27 parents completed the follow-up T3 survey (Figure 1).No significant demographic differences existed between enrolled parents who completed the T2 survey and parents who did not.However, the duration of their child's illness differed between groups, with a median (IQR) illness duration of 7.3 (0.4-20.2) years for parents completing the intervention vs 2.0 (0.9-6.6) years for those who did not (P = .02).

AYA Participation in the PediSICP Intervention
We screened 19 AYAs, of whom 8 were eligible and enrolled (Figure 1).Six AYAs participated in an ACP conversation using the PediSICP framework, 5 AYAs completed the T2 and T3 surveys.One patient disenrolled after the ACP conversation at their parent's request.

Race a,b
African American or Black 2 Hispanic or Latino 10 Not Hispanic or Latino 23 English as first language 27 (81.
a Race and ethnicity are presented as characterized by self-report.
b Multiple selections allowed.

Discussion
The results of this single-group pilot cohort study indicate that the PediSICP was both feasible and acceptable to a variety of stakeholders, including interprofessional clinicians and parents of seriously ill children and provide valuable insights into the benefits of utilizing a structured approach to ACP.
The PediSICP was developed to provide a structured framework for pediatric clinicians without subspecialty palliative care training to engage in effective and efficient ACP.
In the adult population, the efficacy of ACP and its evidence base have been called into question, 41,42 and disagreement remains about the value of ACPs in clinical practice and research. 43,4450][51] Furthermore, this study targeted interprofessional clinicians, which is both novel and aligned with expert recommendations that stress the need to leverage nonphysicians to guide ACP.25,43,45 Another key strength of the PediSICP is its focus on enhancing communication, with most clinicians reporting that they learned something new about and felt more connected to the families they had ACP conversations with, suggesting the program may enhance clinician understanding of family priorities and build trust, which are essential for providing goal-concordant recommendations.
The study also assessed the experiences of parents who engaged in ACP using the PediSICP framework and highlights the potential to enhance patient and family engagement in ACP.Most participating parents reported the intervention was worthwhile and that they would recommend the program to other families, even when these conversations caused sadness or worry.0][61][62] In inquiring about goals and what is most important, clinicians can strengthen alignment and ensure that patients families retain some control despite clinical uncertainty. 63,64The decrease in anxiety levels observed immediately after the intervention and at the 1-month follow-up, from moderate to mild anxiety, aligns with the adult and pediatric advanced cancer literature, highlighting the potential psychological benefits of ACP for families facing serious illness. 26,28,29,61,62,65For therapeutic alliance, to our knowledge, there is no literature in pediatrics describing the minimally important clinical differences in THC scale scores; however, in pediatric advanced cancer populations, similar small statistically significant differences in therapeutic alliance have been reported with advance care planning. 35ture research in the form of an adequately powered randomized clinical trial will attempt to improve on the magnitude of this effect and its duration.

Limitations
This study has several important limitations.The requirement for participants to be English-speaking does not allow for a readily generalizable intervention for diverse populations.Additionally, 97% of the clinicians were White.More research is warranted to understand cultural barriers to ACP, such as race and language, that might impact the program's feasibility.AYAs were underrepresented, as recruitment was limited owing to high medical burden and a large proportion of AYAs with severe neurologic impairment in our sample.Incorporating the voice of children and AYAs is an important future direction for this work.Because participation was voluntary and successful enrollment of clinicians was based on both self-selection and the ability to attend the training, participants may have been predisposed to feel favorably about the intervention.Additional selection bias may have been introduced by having outpatient clinicians serve as referral gatekeepers for some patients.
Given the nature of this pilot study, we did not standardize the time between clinician training and participation in ACP conversations; thus, we were unable to determine whether booster training may be necessary.Furthermore, while we did not detect improvement among some outcome measures, such as quality of communication, as a pilot study, it was intentionally underpowered, and we may have missed identifying improvements in some outcomes.Alternatively, because the QOC questionnaire measures both general communication and communication about EOL care, it is possible that some items were not relevant or that parents or AYAs did not feel connected with the clinicians conducting ACP conversations.

Conclusions
The findings of this cohort study suggest that the PediSICP represents a significant step toward addressing the unmet need for pediatric ACP by providing a framework for clinicians caring for pediatric patients who are seriously ill and their families.The number of children living with serious illness is growing, which requires pediatric clinicians to become proficient in ACP to ensure the care they provide is concordant with patient and family goals and values. 43,66,67By equipping clinicians with the necessary training and tools, the PediSICP promotes meaningful ACP conversations that enhance clinician and family experiences and may improve therapeutic alliance and reduce anxiety.
While findings of this pilot study are promising, future studies will evaluate the efficacy of the PediSICP on family-centered outcomes in a randomized controlled trial.Additionally, assessing the scalability and cost-effectiveness of the program will be essential for its widespread adoption.

JAMA Network Open | Pediatrics
Feasibility of a Serious Illness Communication Program for Pediatric Advance Care Planning

Figure 2 .
Figure 2. Clinician Reactions to Having an Advance Care Planning Conversation Using the Pediatric Serious Illness Communication Program Framework

Figure 3 .
Figure 3. Parent Reactions to Having an Advance Care Planning Conversation Using the Pediatric Serious Illness Communication Program Framework Feasibility of a Serious Illness Communication Program for Pediatric Advance Care Planning JAMA Network Open.2024;7(7):e2424626.doi:10.1001/jamanetworkopen.2024.24626(Reprinted) July 26, 2024 3/13 Downloaded from jamanetwork.comby guest on 07/28/2024 consistent with other feasibility studies among parents of children and adults with serious illness.
Race was classified as African American or Black, White, or other (including American Indian or Alaska Native, Asian, multiracial, and Native Hawaiian or Other Pacific Islander), and ethnicity was classified as Hispanic or Latino or not Hispanic or Latino.Participants could select multiple races and ethnicities.To compare participant characteristics by intervention completion status, continuous variables were tested by Wilcoxon rank sum test and categorical variables by Fisher exact test.Wilcoxon signed-rank tests were used to compare differences between instrument scores for exploratory outcomes for parents at 3 time points, as previously described.Given the low number, we did not perform testing to analyze for differences in AYA instrument scores.P values were 2-sided and considered statistically significant at P < .05.Analyses were conducted using R software version 4.3.1 (R Project for Statistical Computing).Data were analyzed from January 2022 to March 2023.

Table 1 .
details clinician reactions to the ACP conversation using the PediSICP framework.Most clinicians (29 clinicians [97%]) agreed or strongly agreed that they felt prepared for the ACP conversation and the guide was useful; all clinicians reported that they would recommend the framework to colleagues, thus meeting our clinician acceptability targets.Most clinicians (29 clinicians [97%]) agreed or strongly agreed that they learned something new about the family and 28 clinicians (93%) felt more connected to the family after the conversation.Most clinicians (29 Characteristics of Interprofessional Clinician Participants Who Completed the Pediatric Serious Illness Communication Program Intervention Feasibility of a Serious Illness Communication Program for Pediatric Advance Care PlanningParent and Clinician Exploratory OutcomesParents reported significantly higher therapeutic alliance at T2 compared with T1 (HCS mean [SD] score, 57.6[6.4]vs55.3[7.8];P=.03),althoughthisdid not persist at the T3 survey (HCS mean [SD] score, 54.8[8.7];P = .97).There were no significant differences in parent report of shared decision- Figure 1.Flowchart of Participant Recruitment to Pediatric Serious Illness Communication Program (PediSICP) Pilot Study Clinicians A Parents B Adolescents and young adults C 137 Approached via email 40 Enrolled and trained JAMA Network Open.2024;7(7):e2424626.doi:10.1001/jamanetworkopen.2024.24626(Reprinted)July 26, 2024 5/13 Downloaded from jamanetwork.combyguest on 07/28/2024 a Race and ethnicity are presented as characterized by self-report.bMultiple selections were allowed.cOther includes American Indian or Alaska Native, Asian, multiracial, and Native Hawaiian or Other Pacific Islander, based on self-report.JAMA Network Open | Pediatrics

Table 2 .
Demographics of AYA and Parent Participants Who Completed the Pediatric Serious Illness Communication Program Intervention